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Sir,

Horner's syndrome is a well-recognized complication of various thoracic surgical procedures. We present a case of Horner's syndrome that had arisen as the complication of chest drain migration in the management of spontaneous pneumothorax.

Case report

A healthy 25-year-old Chinese male presented to the Accidents and Emergency Department of United Christian Hospital with 3 days’ history of right-sided chest pain and shortness of breath. The patient had no other complaint. There was no history of trauma and no history of cervical or thoracic surgery. Clinical examination revealed a lean, medium-height patient with normal external appearance and pupillary response. Examination of the chest showed signs of right pneumothorax. Chest radiography confirmed the presence of right tension pneumothorax with depressed right hemidiaphragm and left mediastinal shift. Tube thoracostomy was performed in a standard fashion. Immediate postoperative chest radiography showed the tip of chest drain pointing upward and overlapping the posterior part of the right fourth rib (Figure 1a).

Figure 1
figure 1

(a) Tip of chest drain overlapped the posterior part of the right fourth rib on day 1. (b) Tip of chest drain overlapped the inferior edge of the posterior part of the right third rib on day 2. (c) Tip of chest drain overlapped the neck of the right second rib and the tip of the right transverse process of the second thoracic vertebra.

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The patient was admitted for daily chest radiography to monitor the resolution of pneumothorax. On the third postoperative day, the patient started mobilization exercise during which he complained of right eye ptosis. Chest radiography on the same day showed upward migration of the chest drain with the tip overlapping the neck of the right second rib and the tip of the right transverse process of the second thoracic vertebra (Figure 1c). The chest drain was removed on day 4 with resolution of pneumothorax, but the ptosis persisted.

The patient was referred to the Ophthalmology Department. Examination revealed right face anhydrosis, right eye ptosis of 4 mm and anisocoria. The right pupil was 4 mm and the left was 6 mm in diameter (Figure 2). The intraocular pressure was 12 mmHg in the right eye and 16 mmHg in the left eye. Exophthalmometer reading was 14 mm for the right and 15 mm for the left eye, respectively. Other ocular examinations including extraocular movement were unremarkable. The upper limb and cranial nerves neurology were normal. Instillation of cocaine 4% confirmed right Horner's syndrome, while pharmacological localization with adrenaline 1 : 1000 showed no denervation hypersensitivity. Repeated radiography including lordotic view of the chest, cervical spine, CT and MRI of the brain was normal. At 2 months follow-up, the right Horner's signs persisted.

Figure 2
figure 2

Right Horner's syndrome: ptosis and anisocoria.

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Comment

In 1869, Swiss ophthalmologist Johann Friedrich Horner described the clinical findings (interruption of cervical sympathetic pathway, ipsilateral miosis, partial ptosis, enophthalmos and anhydrosis), which later became Horner's syndrome. This oculosympathetic palsy may result from any interruption of the three-neuron pathway in its course from posterior hypothalamus to the eye.

There have been several reports of Horner's syndrome associated with spontaneous pneumothorax.1,2,3,4,5 They all had in common: (1) early presentation—during or even before the onset of the chest symptoms and (2) reversibility upon relief of pneumothorax. The underlying pathogenesis was presumably because of traction on the sympathetic fibres in superior mediastinal displacement.2,3,4,6

Horner's syndrome has also been reported as complication of tube thoracostomy in the management of various pneumothoraxes (traumatic, spontaneous and iatrogenic) and after thoracic operations.6,7,8,9,10,11,12 One common feature was the radiographic close proximity between the tip of chest drain and the lung apex in the initial insertion. Direct trauma, local haematoma, pressure ischaemia, inflammation and induced adhesion were all suggested to play a role.6,7,9 The onset of Horner's syndrome after tube thoracostomy varied considerably from 12 hours postinsertion to 42 days after the removal.7,10 In total, 57% of cases showed no signs of reversibility after a follow-up period from 3 months to 1 year despite removal of the chest drain.6,7,9,12 While there was no association between the timing of onset and reversibility, for those with repositioning within 1 day, 66% of cases showed recovery of the Horner's signs.

In our patient, we believed that the second-order preganglionic neuron was injured by the migrating chest drain during its course from the ciliospinal centre of Budge to the superior cervical ganglion near the apical pleura. To avoid injury of the apical structure, chest drain insertion of less than 10 cm in the thoracic cavity and inferior to the second rib posteriorly has been suggested.9,10,12 In addition, we suggest: (1) firm securing of chest drain to skin with large sutures (0 ‘O’ silk) and adhesive tape, (2) daily radiographic monitoring of the chest drain position together with clinical examination of the lid position and pupillary response is necessary when chest drain is in situ, and (3) prompt repositioning of any chest drain with significant upward migration to prevent irreversible iatrogenic Horner's syndrome.